• Jinxin Yang, Qianlei Liang, Liang Han, Yan Wang, and Yongchuan Guo.
• Department of Neurosurgery, The Second Hospital of Jilin University, Changchun, China.
• Medicine (Baltimore). 2024 Apr 19; 103 (16): e37869e37869.

RationaleEosinophilic angiocentric fibrosis (EAF) is considered to be a kind of benign IgG4-related disease, and it is more often found in the nasal cavity. We present a pretty rare case of orbital EAF that is unlike any other reported case for this case is an IgG4 negative orbital EAF and successfully treated by the fronto orbitozygomatic approach surgery.Patient ConcernsThis is a 68-year-old man from a rural area of Inner Mongolia Autonomous Region, went to our hospital for a 2-month history of vision loss with a local hospital orbital computer tomography which showed that there was a lesion in his left orbit. The inspection of the patient revealed that the patient left eye was protruding outward and the left eyelid unable to complete open or close. And his left eyeball movement had difficulty in all directions. Postoperative pathology diagnosed that this was a case of IgG4-negative EAF case.DiagnosesOrbital EAF.InterventionsSurgical radical resection and postoperative glucocorticoid therapy.OutcomesAfter surgery, the left eye vision of this patient increased to 0.6 tested in the standard logarithmic visual acuity chart. And his left eyeball movement dysfunction and eyeball outward protruding get a partially relief.LessonsEAF occurring in the orbit is a very rare disease and immunohistochemical results of EAF can be IgG4 negative.Copyright © 2024 the Author(s). Published by Wolters Kluwer Health, Inc.

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